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   Table of Contents - Current issue
Coverpage
January-June 2019
Volume 6 | Issue 1
Page Nos. 1-64

Online since Monday, February 4, 2019

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EDITORIAL  

Evolution of cleft orthodontics in India p. 1
Ashok Utreja
DOI:10.4103/jclpca.jclpca_37_18  
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STATE OF THE ART Top

Reoperation of bilateral cleft lip deformity after primary Manchester or Veau III repair p. 3
Maurice Y Mommaerts, Ana Tache, Natalie A. J. Loomans
DOI:10.4103/jclpca.jclpca_36_18  
Residual bilateral cleft lip deformities are commonly attributed to neglect of oral sphincter reconstruction during primary repair. This report describes a straightforward technique for reoperation that is applicable for most patients. It also describes the results of a retrospective observational cohort study of 21 patients that underwent reoperation performed by the senior author (MYM) from 1991 to 2018. The patients had undergone primary lip repair at other institutions. Their mean age at reoperation was 17 years. Pre- and post-operative photographs (at 3.5 years' postoperation) were assessed for 15 parameters. Overall configuration, scar appearance, philtrum anatomy, Cupid's bow shape, and esthetic aspects of lip dynamics improved considerably. This procedure is the same as that used by the authors in 4-month-old children who have undergone a nasolabial adhesion procedure at 4 weeks of age. We discuss the rationale of using this procedure for primary lip repair, thus eliminating the need for reoperation.
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ORIGINAL ARTICLES Top

Correlating causative factors in cleft lip and palate patients: An epidemiological study p. 11
Bhagyashree B Desai, Dolly P Patel, Surina V Sinha, Mahesh Jain, Roopal N Patel, Sheron T Bhanat
DOI:10.4103/jclpca.jclpca_12_18  
Aims: The primary aim of this study was to correlate the incidence of cleft lip and palate (CLP) with possible etiological factors such as consanguinity, history of abortions or miscarriages, type of delivery, oral destructive habits, and family history. Materials and Methods: This retrospective study was completed using a standardized questionnaire which was prepared to investigate the information of 166 patients referred to/undergoing orthodontic rehabilitation treatment of CLP at the hospital. The variables assessed were consanguinity, affected members in family and relatives, gestational history, past abortions and/or miscarriages, medications taken by mother during pregnancy, and parental habits of tobacco chewing and/or smoking. Statistical Analysis Used: Correlation of the above-mentioned variables as well as any mutual effects of gender and cleft type was assessed statistically by Pearson's Chi-square test and Fisher's exact test. Results: The results of the present study provide demographic details of cleft patients in Gujarat, which show that cleft deformity is seen more in male patients (n = 98; 59%). Unilateral CL and palate of left side (CL + PUL) occurs most frequently (n = 62; 37.3%) both in male (n = 40; 40.8%) and female (n = 22; 32.4%) patients. Occurrence of CL + PUL is followed by bilateral CL and palate (CL + PB) (n = 45; 27.1%) and unilateral CL and palate of right side (n = 27; 16.3%). The oral destructive habits of parents (n = 82; 49.4%), past abortions and miscarriage (n = 47; 28.3%), family history (n = 26; 15.7%), and consanguinity (n = 24; 14.5%) could be correlated with the occurrence of CLP. Conclusion: This can serve as a guide for future reference to health workers so that they can take measures to create awareness among the people to avoid consanguineous marriages, use of tobacco, abortions, and create awareness about a family history. The outcome could also help government/public health sector workers to develop strategies for awareness, identification, and treatment of cleft deformities.
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Use of hyrax distractor in maxillary hypoplasia associated with cleft lip and palate patients p. 17
Neeraj Upadhyay, Arun Kumar Singh, Veerendra Prashad, Divya Narayan Upadhyaya
DOI:10.4103/jclpca.jclpca_33_18  
Context: Use of hyrax expander for palatal arch lengthening (anteroposterior) is not new, but large series with long-term follow-up is lacking. Aims: The aim of this study was to evaluate surgically assisted rapid maxillary expansion for hypoplastic maxilla, its efficacy, patient's satisfaction, and relapse rates. Settings and Design: A prospective study was done in PGD of Plastic Surgery, KGMU, Lucknow, between October 2014 and December 2015. Subjects and Methods: Fifteen patients with maxillary hypoplasia associated with cleft lip and palate underwent anterior segmental maxillary osteotomy. Distraction of anterior segment was done with a tooth-anchored hyrax expansion screw oriented in anteroposterior direction. Distraction was started on the 7th day. After desired advancement, the device was left intact for a consolidation period of 3 months and follow-up for any complication was done at the end of 6 months. Statistical Analysis Used: IBM SPSS version 20 software was used. Data expressed as number (percentage) and mean ± standard deviation, Student's t-test and ANOVA were used to compare mean between groups. Results: Bony landmarks sella–nasion–A point angle, maxillary length (ANS-PNS), mean space created on one side (mesial border of first molar to distal border of first premolar), and point A-N perpendicular improved significantly (P < 0.001). There was no significant change in vertical height of maxilla. Soft-tissue profile of patient (Sn–G perpendicular) also improved significantly (P < 0.001). No relapse was observed in patient during the 6 months of follow-up. Conclusion: Results of hyrax distractor in maxillary hypoplasia associated with cleft lip and palate patients were highly promising in patients with occlusal discrepancy <8 mm.
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Endoscopic-assisted craniosynostosis surgery: First case series in India p. 23
Derick A Mendonca, Venkat Ramamurthy, Swaroop Gopal, Rajendra S Gujjalanavar
DOI:10.4103/jclpca.jclpca_28_18  
Introduction: Craniosynostosis is a congenital pathological condition resulting from premature fusion of sutures of the cranial vault and leads to an abnormal head shape with a significant risk of raised intracranial pressure. Surgical correction techniques have seen a constant evolution from an extensive open procedure to the relatively new minimally invasive craniosynostosis technique. The current center introduced this new procedure in India since 2015. Aims and Objectives: This article documents our experience as the first team in the country to correct craniosynostosis endoscopically with emphasis on planning, surgical techniques, and helmet therapy to achieve the optimum result. Materials and Methods: This is a prospective case series of six patients with anterior craniosynostosis corrected endoscopically and followed up with postoperative custom-made cranial helmet. All patients were nonsyndromic with no associated anomalies. The mean age of surgical intervention was 92.6 days (84–100 days), mean duration of surgery was 61.7 min (54–74 min), mean blood loss was 55 ml (50 ml–60 ml), and mean duration of hospital stay was 2 days (2–3 days). No complications and mortality were reported. Conclusion: This case series has demonstrated that endoscopically assisted craniosynostosis correction is a safe, effective, and reliable technique in the armamentarium of a craniofacial surgeon. The minimally invasive nature allows early intervention in children. Parental compliance is important in helmet therapy which forms an integral part of this surgery for correction of the abnormal head shape. We believe that every patient with nonsyndromic single-suture craniosynostosis should be offered endoscopic correction before the age of 6 months.
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Association of cleft lip and/or palate in people born to consanguineous parents: A 13-year retrospective study from a very high-volume cleft center p. 33
Praveen Kumar Neela, Srinivas Gosla Reddy, Akhter Husain, Vasavi Mohan
DOI:10.4103/jclpca.jclpca_34_18  
Objective: The objective of this study was to investigate the association of cleft lip (CL) and/or cleft palate (CP) in people born to consanguineous parents. Methodology: This retrospective study was conducted at GSR Institute of Craniofacial Surgery, Hyderabad, a very high-volume cleft center. Medical records of 18,242 cleft patients who came for treatment in the past 13 years were physically searched for the presence of consanguinity of their parents. About 3653 patients' case records were identified. Then, the data related to gender, type of cleft, degree of consanguinity, presence of positive family history were collected from these 3653 patients' records and entered in Microsoft Excel which was later sent for statistical analysis. Results: Distribution of study participants according to various study variables such as gender, type of cleft, and degree of consanguinity showed interesting findings. Almost 20.02% of the total 18,242 cleft patients' records showed consanguinity of their parents. Nearly 96.7% of patients had CL. CP was seen in 73.7% of patients. About 57.8% of parents had the first degree of consanguinity. Only 3.5% had a positive family history. Chi-square test showed that there was a significant difference in the distribution of patients according to degree of consanguinity and CL, variations in CL and gender, cleft alveolus and gender, degree of consanguinity and CL. Conclusion: Consanguinity is a major etiological factor in CL and/or CP.
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To assess the prevalence of dental caries and to evaluate oral health-related quality of life in cleft lip and palate patient p. 38
Amit Manohar Reche, Shrutika Dhanrajji Chordiya, Sakshi Pradeep Joshi, Nikita Suresh Bhumbre, Shivani Kishore Jadhav, Nikita Pramod Dhange
DOI:10.4103/jclpca.jclpca_24_18  
Aim: The aim of this study was to assess the prevalence of dental caries and evaluate oral health-related quality of life (QoL) in patients with cleft lip and palate (CLP) in Vidarbha region. Settings and Design: A cross-sectional questionnaire study was conducted in the Department of Public Health Dentistry of our institute. The sample was divided into three groups. Subjects and Methods: Group A (110) patients with unilateral CLP (UCLP), Group B (110) patients with bilateral CLP (BCLP), and Group C (150) control group. From self-administered questionnaire, the patients were asked 10 questions based on eating, speaking, toothbrushing, sleeping, smiling, emotional status, carrying out work, and social contact and rated as 0, 1, 2, and 3 (no problem, mild, moderate, and severe). Statistical Analysis Used: ANOVA and Pearson's correlation tests were performed. Results: According to the results, there is no statistical difference for decayed, missing, and filled teeth score seen between UCLP and BCLP patients; however, there is significant difference (<0.001) seen between the cleft patients and control group. According to questionnaire result, the patients with bilateral cleft experienced more difficulty while eating, speaking, smiling, and maintaining social contact as compared to the control group. Conclusion: Patients suffering from CLP showed higher caries prevalence compared to the control group; on the other hand, negative impact has been seen on the QoL of CLP patients.
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Orofacial cleft patient referral to a university orthodontic clinic over a 20-year period in Ghana p. 43
Merley Afua Newman-Nartey, Gwendolyn N Amarquaye, Kwabena Gyaami Amoah, Alexander Oti Acheampong, Nii Otu Nartey
DOI:10.4103/jclpca.jclpca_17_18  
Context: After the first orofacial cleft (OFC) patient was referred to the orthodontic clinic of the University of Ghana School of Medicine and dentistry in 1998, a multidisciplinary clinic at the Korle-Bu teaching hospital (KBTH) was inaugurated and partnered by non-governmental organization, Transforming Faces from 2003 to 2014 and Smile Train from 2014 to date. Aims: The aim of this study was to review and document presentations, management, and associated care influences of OFC patients referred for orthodontic care to influence quality improvement policy. Settings: This study was conducted at the Orthodontic Clinic of the University of Ghana School of Medicine and Dentistry. Design: This was a retrospective design, in which data from patient records were obtained and analyzed. Subjects and Methods: Review of records of all OFC patients who assessed the orthodontic clinic from 1998 to 2018. Statistical Analysis Used: The statistical analysis was done using Microsoft Excel. Results: Thirty-four patients were referred to the orthodontic clinic from 1998 to 2018. The majority (67.65%) was referred from the KBTH multidisciplinary clinic and smaller number (32.35%) from other hospitals and clinics. Their ages ranged from 2 weeks old to 20 years old. The majority of the referred patients were in the 5–9-year-old age group (35.29%). Of the 26 patients who underwent treatment, the majority 38.46% were treated using the less expensive fixed upper-only appliance option and obturators were not routinely used (15.38%). The majority of patients were self-funded (47.8%) and (34.8%) were pro bono cases of which over 50% of discontinued treatment. Conclusions: The multidisciplinary OFC clinic was beneficial as the major source of orthodontic referrals. More stringent criteria for the selection of pro bono cases could decrease loss from care and inclusion of complete OFC care in the National Health Insurance Scheme may remove financial barriers.
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Risk indicators for syndromic and nonsyndromic orofacial clefts in Southern Province of Saudi Arabia p. 51
Bandar Alyami, Mutaz Ali-Hassan, Manasaour Al-Mahri, Fahad Alyami, Saeed Alharieth
DOI:10.4103/jclpca.jclpca_31_18  
Objective: This study was designed to find out risk indicators for the development of orofacial clefts (OFCs) in the Southern Province of Saudi Arabia. Materials and Methods: This was a retrospective study carried out in a maternity hospital from January 2013 to December 2016. Data including child sex, type and site of cleft, birth weight and birth month (season), parents' age at the time of their child's birth, paternal and maternal habits, parental consanguinity, and family history of OFCs were obtained. The data were processed and analyzed using SPSS software, Version 20. Results: Overall prevalence of OFCs in the study population was 0.65/1000 live births. There were 10 males, 5 females, and 1 ambiguous sexual orientation. Of the 16 OFC patients, 9 (56.25%) were classified as having syndromic OFCs (SOFCs) and 7 (43.75%) as having non-SOFCs (NSOFCs). Cleft lip and cleft lip/palate both had strong family history of OFCs. Similarly, the phenotype of OFCs and the presence of syndromic or NSOFCs did not show a relationship with paternal smoking habits during pregnancy. No relationship was reported between parental consanguinity with the presence of these factors. However, results showed that 13 (81.3%) out of 16 identified cases of OFC had background consanguinity. Conclusion: Prevalence of OFC in the current study was within reported range, consanguinity was observed in 81.3% of identified cases. Further research is paramount in the role of consanguinity in OFC in Arabian population.
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CASE REPORTS Top

Median cleft lip – A case report of rare congenital anomaly p. 56
Abhay N Datarkar, Jagadish U Patil
DOI:10.4103/jclpca.jclpca_30_18  
Median cleft lip is a midline vertical cleft through the upper lip in the absence of a prolabial remnant. This is a rare anomaly with very few cases described in the literature. This may occur as a sporadic event or as a part of an inherited sequence of anomalies. It arises embryologically from incomplete fusion of the medial nasal prominences. The author presents a rare case of median cleft lip, broad nose, and wide ala with hypertelorism. The embryology, presentation, and surgical technique for treating these cases are discussed.
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Beard hemangioma associated with cleft lip and palate: An unusual case and literature review p. 59
Nicolás E Solano, Ejusmar K Rivera, Salomon Ramos, Maholy Sánchez
DOI:10.4103/jclpca.jclpca_27_18  
Infantile hemangiomas (IHs) are the most common benign vascular tumor of infancy. Sixty percent of lesions affect the head and neck. There are special considerations in IHs, like beard-distribution hemangiomas, in which preauricular areas, chin, anterior neck, and lower lip are involved and they have been associated with the presence of airway hemangiomas. In contrast, labial and palatal clefts are the most common congenital malformations of the craniofacial region. The etiology is multifactorial and includes both genetic and environmental factors. It is associated with more than 500 syndromes with varying levels of penetrance and expressiveness. IHs and cleft lip and palate (CLP) are relatively common conditions found in newborns. Even though they are common enough, there is no evidence reporting a patient with both entities simultaneously. The purpose of this study is to present an unusual case of beard hemangioma in a 3-month-old female baby associated with CLP.
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CLINICAL INNOVATION Top

Putty-based surgical site protection during intraoral trimming of the hyrax distractor tooth borne appliance p. 62
Tulika Tripathi, Navneet Singh, Priyank Rai, Prateek Gupta
DOI:10.4103/jclpca.jclpca_23_18  
Background: HYDIS-TB is a contemporary distraction appliance used in large alveolar cleft defects. This appliance requires intraoral trimming at the docking site during approximation of the segments which cause entrapment of acrylic particles at the surgical site thereby hampering normal healing process. Solution: We employed commonly available putty material at distraction site in a cleft patient during intraoral acrylic trimming of the appliance. This shielded the surgical site from exogenous material, which facilitated proper healing.
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